Download A 35-year-old male with chronic cough CASE FOR DIAGNOSIS

Eur Respir J 2007; 29: 608–611
DOI: 10.1183/09031936.00099706
CopyrightßERS Journals Ltd 2007
CASE FOR DIAGNOSIS
A 35-year-old male with chronic cough
C. Moschos, I. Kalomenidis, C. Roussos and G.T. Stathopoulos
CASE REPORT
A 35-yr-old male was examined in the outpatient clinic of the
present authors’ department. He complained of a dry paroxysmal cough of indeterminate origin that he had noticed 3
months earlier. The cough was associated with a tickling feeling
in the throat. There was no association between coughing flares
and meals, nasal discharge, season, time of day or body
posture. The patient did not recall having symptoms compatible with an upper airway viral infection in the past 3 months
and reported no gastro-intestinal symptoms like daily heartburn and regurgitation. He lived in the city, was a lifetime
nonsmoker, his previous medical history was unremarkable
and he was not taking any medication. He worked as an
electronic engineer in a smoke- and dust-free environment and
exhibited a type A personality. Upon appearance of the cough
3 months earlier, he had been examined by a physician and had
since received inhaled salmeterol (50 mg b.i.d.) and fluticasone
(250 mg b.i.d.), without improvement.
A thorough physical examination and routine laboratory tests,
including white blood cell count and differential, red blood cell
count, erythrocyte sedimentation rate, liver and renal function
tests, serum C-reactive protein and an ECG were normal. Chest
(fig. 1) and paranasal sinus (image not shown) radiographs
were taken. Ear, nose and throat assessment was unremarkable.
After discontinuing treatment with inhaled salmeterol and
fluticasone, simple spirometry pre- and post-bronchodilator
(four puffs of 100 mg salbutamol) and bronchoprovocation
challenge with methacholine were performed (fig. 2). The
patient was given a peak flow meter and during a monthly
observation period his diurnal peak flow variation was below
5% (data not shown).
a)
FIGURE 1.
b)
Chest radiograph showing a) posterior-anterior view and b) lateral view.
Dept of Pulmonary and Critical Care Services, General Hospital ‘‘Evangelismos’’, School of Medicine, National and Kapodistrian University of Athens, Athens, Greece.
STATEMENT OF INTEREST: None declared.
CORRESPONDENCE: G.T. Stathopoulos, Dept of Pulmonary and Critical Care Services, General Hospital ‘‘Evangelismos’’, School of Medicine, National and Kapodistrian University of Athens, 3 Ploutarhou Str.,
10675 Athens, Greece. Fax: 30 2107235521. E-mail: [email protected]
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C. MOSCHOS ET AL.
b)
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5
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+
c)
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ll
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4
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6
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Methacholine dose mg
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FEV1 L
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0
-2
-4
-6
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-10
-12
-14
Volume L
Flow L·s-1
a)
A 35-YEAR-OLD MALE WITH CHRONIC COUGH
3
2
2
1
0
FIGURE 2.
1
2
3
4
Volume L
5
6
7
0
0
0.5
1
1.5
2
2.5
Time s
0
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Pulmonary function tests of a) flow–volume plot and b) volume–time curve. –––: pre-bronchodilator; ????: post-bronchodilator; #: predicted value. c)
Methacholine dose–forced expiratory volume in one second (FEV1) curve. #: peak expiratory flow rate; ": forced expiratory flow (FEF) at 50% of forced vital capacity (FVC); +:
FEF at 75% of FVC; 1: FEV1; e: FVC.
a)
FIGURE 3.
b)
a and b) Computed tomographic scans of the chest.
Since an empiric trial of inhaled corticosteroids had already
been administered, treatment was discontinued and empiric
treatment for gastro-oesophageal reflux disease was administered with a proton pump inhibitor (omeprazole, 20 mg b.i.d.)
over a period of 2 months. Neither withholding inhaled
steroids nor omeprazole administration had any effect on the
patient’s symptoms and coughing persisted. Therefore, a
computed tomographic (CT) scan of the chest was ordered
(fig. 3), followed by transthoracic echocardiography (fig. 4).
FIGURE 4.
Transthoracic ultrasound.
EUROPEAN RESPIRATORY JOURNAL
BEFORE TURNING THE PAGE, INTERPRET
THE CHEST RADIOGRAPHS, PULMONARY
FUNCTION TESTS, COMPUTED TOMOGRAPHY AND ULTRASOUND, AND SUGGEST
A DIAGNOSIS.
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c
A 35-YEAR-OLD MALE WITH CHRONIC COUGH
INTERPRETATION
Chest radiography
Figure 1 shows near-normal normal chest radiographic films.
After careful evaluation, and with some difficulty, a round,
homogeneous opacity can be identified in the lateral projection, located well above the diaphragm and superimposed on
the cardiac opacity.
Pulmonary function and bronchoprovocation challenge
tests
Figure 2a and b show normal flow–volume and volume–time
curves, respectively, with no response to bronchodilation.
Figure 2c shows a normal forced expiratory volume in one
second (FEV1)–methacholine dose plot, without significant
bronchoconstrictor response (,8% drop in baseline FEV1),
even at maximal methacholine dosage (2 mg).
Computed tomography
The CT images in figure 3 (soft-tissue gating (a); and lung
parenchymal gating (b)) show a solitary, unilocular, thinwalled, round cyst, sized 364 cm, situated along the left
inferior cardiac border, exhibiting an attenuation coefficient
between those of water and fat. No other lesions were seen.
Transthoracic ultrasound
Transthoracic ultrasonography showed a cyst measuring
364 cm in close association with the pericardium, and ruled
out any haemodynamic changes, cardiac compression, right
ventricular outflow obstruction or atrial enlargement.
Diagnosis: Left-sided pericardial cyst.
CLINICAL COURSE
After consultation with the patient on the possibility that his
cough may be caused by the pericardial cyst, a decision was
made to proceed to surgical removal of the cyst. Pre-operative
bronchoscopy, performed to rule out coexisting endobronchial
lesions, was unremarkable. The cyst was thereafter removed
via video-assisted thoracoscopic surgery. Pathological examination of the cyst concurred with the diagnosis of pericardial
cyst. The patient’s cough resolved immediately after surgery
and has not reappeared after 6 months of follow-up.
DISCUSSION
Cough is the most common respiratory symptom encountered
by clinicians [1]. Acute cough is usually easier to diagnose and
resolves after proper therapy. Chronic cough is defined as
cough lasting .8 weeks, and can be a significant problem,
interfering with important daily activities. The most common
causes of chronic cough are asthma, gastro-oesophageal reflux
disease, upper airway cough syndrome, nonasthmatic eosinophilic bronchitis or a combination of the above [2]. Although
some authorities suggest that the cause of chronic cough
can be identified in up to 95% of cases, in ,20% of patients a
diagnosis cannot be established even after ruling out uncommon causes of chronic cough [1]. Diagnostic tests limited to the
respiratory system do not always provide a diagnosis, since
cough can be the major or presenting symptom in a large
percentage of patients with common and uncommon nonpulmonary diseases. Mediastinal lesions commonly cause
cough by impinging the adjacent airways or the vagal afferent
nerves that regulate involuntary coughing, but cough is
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C. MOSCHOS ET AL.
rarely the main or sole manifestation. The most common
mediastinal lesions that can cause cough are oesophageal cysts
and tumours, lymphomas, mediastinal lipomatosis, and
neuromas and neurilemmomas of the vagus or internal
laryngeal nerves [2].
Pericardial cysts are the second most common type of primary
mediastinal cysts after bronchial cysts and constitute 7% of all
mediastinal lesions [3]. They are most frequently congenital,
arising from aberrant fusion of pericardial lacunae in
approximately one in 100,000 individuals; however, some
may be acquired. They remain unchanged in size or slowly
enlarge over a period of many years and are often discovered
in routine chest radiographs of asymptomatic adults, usually
in the fourth to fifth decade of life. They are usually unilocular
cystic lesions with a wall of varying thickness with fat cells,
lymphocytes, blood vessels and muscle fibres and clear fluid
contents, hence the name ‘‘clear water’’ or ‘‘spring water’’ cyst
[4]. Of all pericardial cysts, 70% are located in the right
cardiophrenic angle, 22% in the left cardiophrenic angle and
the remaining 8% elsewhere in the pericardium (e.g. the
posterior mediastinum, the right or left hilar region, the right
paratracheal area or the para-aortic region) [3]. The differential diagnosis of pericardial cyst includes foramen of
Morgagni diaphragmatic hernia, large right pericardial fat
pad, mediastinal or diaphragmatic tumours, and tumours of
the heart or pericardium [5]. Thymomas are a relatively
common cause of mediastinal mass in young patients, and
while thymoma usually resides just anterior to the aortic root,
it can occur anywhere from the neck to the cardiophrenic
angle. Most thymoma patients are asymptomatic, although
one third experience chest pain, cough, dyspnoea and/or
other symptoms related to compression or invasion of
adjacent structures [6]. Although the majority of patients
with pericardial cysts are asymptomatic, about one third
exhibit symptoms.
The most common symptoms associated with pericardial cysts
are vague chest pain, dyspnoea and persistent cough [7–9].
However, complications can be life-threatening, including
cardiac compression, right ventricular outflow obstruction,
cyst rupture with cardiac tamponade and cyst infection with
cardiac or large vessel erosion. There have been reports of
pericardial cysts causing atrial fibrillation by obstructing right
atrial filling or even sudden death after a stress test, and of
hydatid pericardial cysts presenting with circulatory collapse
[5, 10–12]. There are no reports of malignant transformation.
Traditional treatment includes thoracotomy or thoracoscopic
removal, but pericardial cysts can also be diagnosed and
treated by percutaneous aspiration and injection of ethanol as a
sclerosing agent with no evidence of recurrence upon a 3-yr
follow-up [9]. Recurrence has been reported after thoracoscopic removal with successful thoracoscopic re-excision [13].
In the present case, after ruling out common causes of chronic
cough according to current guidelines, the diagnostic evaluation suggested that an uncommon extrapulmonary cause,
such as a left-sided pericardial cyst, was responsible for the
patient’s cough, probably via stimulation of the afferent vagal
receptors from the airways [14]. In this regard, the tickling
sensation in the patient’s throat associated with the cough,
disappeared with the cough after removal of the pericardial
EUROPEAN RESPIRATORY JOURNAL
C. MOSCHOS ET AL.
A 35-YEAR-OLD MALE WITH CHRONIC COUGH
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cyst. As hydatid disease is endemic in Greece, antibodies
against Echinoccocus granulosus were measured in the patient’s
serum but were not detectable.
In conclusion, although it is rare that a pericardial cyst should
present with the sole symptom of cough, it should be
considered in the differential diagnosis of patients with chronic
cough after excluding other common causes.
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